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Case Reports in Medicine

Volume 2017, Article ID 9483738, 4 pages
https://doi.org/10.1155/2017/9483738

Case Report
Ameloblastic Fibroodontoma: Uncommon Case Presentation in
a 6-Year-Old Child with Review of the Literature

Anshad Mohamed Abdulla,1 G. Sivadas,2 L. K. Surej Kumar,3

C. S. Sheejith Hari Peeceeyen,4 and Vaishnavi Vedam5
1
Department of Paedodontics and Preventive Dentistry, International Medical University (IMU), Kuala Lumpur, Malaysia
2
Department of Paedodontics and Preventive Dentistry, Faculty of Dentistry, Asian Institute of Medicine,
Science & Technology (AIMST) University, Kedah Darul Aman, Malaysia
3
Department of Oral and Maxillofacial Surgery, KIMS Hospital, Trivandrum, Kerala, India
4
Dental Services, KIMS Hospital, Trivandrum, Kerala, India
5
Department of Oral Pathology, Faculty of Dentistry, Asian Institute of Medicine, Science & Technology (AIMST) University,
Kedah Darul Aman, Malaysia

Correspondence should be addressed to G. Sivadas; sivadas@aimst.edu.my

Received 31 May 2017; Accepted 16 July 2017; Published 13 August 2017

Academic Editor: Benedetto Bruno

Copyright © 2017 Anshad Mohamed Abdulla et al. This is an open access article distributed under the Creative Commons
Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is
properly cited.

Ameloblastic fibroodontoma is a benign mixed odontogenic neoplasm considered in patients with asymptomatic swelling and
unerupted teeth that exhibit histologic features between ameloblastic fibroma and complex odontoma. Radiographically, this lesion
appears as radiolucency admixed with focal radio opaque masses of irregular shapes and sizes. This lesion is confirmed by the
presence of proliferating odontogenic epithelium, ectomesenchyme, and dental hard tissue formation on pathological analysis
supplementing clinical and radiographic findings. As this tumour is less commonly seen in routine clinical practice, ameloblastic
fibroodontoma with detailed orofacial features and periodic approach to its diagnosis is discussed. This paper reports a case of
ameloblastic fibroodontoma of the mandible in a 6-year-old male patient with an uncommon case presentation and review of the
literature.

1. Introduction We present a case of ameloblastic fibroodontoma (AFO)

Odontogenic tumours are a diverse group of diseases ranging
from simple hamartomas to neoplasms with metastatic po-
tential [1]. Ameloblastic fibroodontoma (AFO) is a rare tu-
mour characterized by proliferating odontogenic epithelium
within primitive cellular ectomesenchymal tissue with/with-
out hard tissue formation [2]. This tumour is characterized
by well circumscribed, asymptomatic slow expansile swelling
that is usually associated with unerupted or displaced tooth
[3]. Initially it was termed as ameloblastic odontoma, but due
to its exceptional mixed nature of growth and incidence of
two types of odontogenic tumours with diverse histological
and biologic behavior, World Health Organization (WHO)
suggested this term to be inappropriate. Later, it was aptly
named as ameloblastic fibroodontoma (AFO) by Hooker
(1967) [4].
in a 6-year-old male patient with distinctive orofacial man-
ifestations, radiographic findings, histopathological report,
and differential diagnosis with treatment to add to the
existing knowledge.
2. Case Presentation
A 6-year-old male patient reported to the dental hospital
with a chief complaint of swelling on the right side of face
since 1 month. History revealed a slow growing and non-
tender swelling. Medical, personal, and family history was
noncontributory. The patient was well oriented with stable
vital signs. On extraoral examination, mild facial asymmetry
due to swelling in the right body of mandibular region, with
ill-defined margins, was evident. On palpation the swelling
was asymptomatic and hard in consistency. Skin over the
2 Case Reports in Medicine
Figure 1: Intraoral examination reveals the obliteration of buccal
sulcus adjacent to molar teeth with buccal and lingual cortical
expansion.
Figure 2: Orthopantomogram reveals radiopaque lesion sur-
rounded by a radiolucent zone extending anteriorly from the lower
first molar region to body of the mandible posteriorly on the right
side.
swelling was normal with no evidence of secondary changes.
Restricted temporomandibular joint (TMJ) movements and
palpable lymph nodes were evident.
On intraoral examination, obliteration of the buccal sul-
cus in molar region with buccal and lingual osseous cortical
expansion was apparent (Figure 1). On palpation, the swelling
was fluctuant, nontender, and fixed to the underlying bone.
Orthopantomogram (OPG) exhibited a large radiopaque
lesion surrounded by a radiolucent zone, which extended
anteriorly from the lower first molar region to body of the
mandible posteriorly on the right side (Figure 2). Routine
blood investigations were also under normal limits which
ruled off evidence of any systemic disorder.
Based on clinicoradiographical findings, a provisional
diagnosis of complex odontoma was made. The patient
underwent enucleation of the lesion and curettage under
general anesthesia. Tooth attached to the follicle around the
lesion was removed. The cavity was irrigated carefully and the
debris was detached. The flap was repositioned in the same
location and suturing was done with vicryl 3.0 suture material
(Figures 3(a) and 3(b)).
Surgical specimen was fixed in 10% formalin and sub-
jected to pathological analysis. On gross examination the
specimen consisted of a hard tissue mass with a soft tissue
attachment. Routine histopathological examination revealed
strands and islands of odontogenic epithelium with periph-
eral palisading nuclei resembling ameloblast-like cells and
loosely arranged central cells, identical to stellate reticu-
lum, embedded in a myxoid cell-rich stroma resembling
the dental papilla. Dentin and enamel were also present
(Figures 4(a) and 4(b)). Correlating the clinicoradiological
and histopathological findings the lesion was diagnosed as
ameloblastic fibroodontoma. Follow-up with this patient
2 years postoperatively exhibited good prognosis and no
evidence of recurrences.
3. Discussion
Ameloblastic fibroodontoma has been conventionally clas-
sified as a benign mixed odontogenic tumour with limited
cases documented in literature. This lesion is reported in
approximately 0.3% to 1.7% of odontogenic tumours among
different locations with 4.6% accounting to total number of
paediatric cases. Children or teenagers with a mean age of
8–12 years are commonly affected with no obvious gender or
anatomic site predilection. This lesion is frequently seen in
the posterior border of the mandible (2.4 : 1) as in the present
case [5].
Debate exists concerning the histogenesis of the amelo-
blastic fibroodontoma as a mixed odontogenic neoplasm.
Previously, concept stated this lesion to be continuum of
differentiation from ameloblastic fibroma to complex odon-
toma. However, the recent literature stated that ameloblastic
fibroodontoma as a discrete entity but it can be histologically
indistinguishable from immature complex odontoma due to
the presence of abundant dental hard tissue formation [6].
According to the revised World Health Organization (WHO)
classification, ameloblastic fibroodontoma is now considered
as a benign tumour without invasive growth [7]. Philipsen et
al. suggested the reduced mean age of AFO, relative arrange-
ment of the soft tissues, and the stage of development of the
involved tooth as the key indicative factors of differentiation
between ameloblastic fibroma and fibrodentinoma [8, 9]. Pre-
dominantly AFO appears hamartomatous while few others
appear to have true neoplastic nature [10].
Patients present with swelling, unerupted tooth, rapid
destruction, and cortical plate expansion. This case also pre-
sented with marked intraoral and extraoral swelling result-
ing in the obliteration of molar buccal sulcus and cortical
plate expansion. World Health Organization (WHO) also
described this well encapsulated tumour to be a benign odon-
togenic neoplasm admixed in primitive ectomesenchymal
tissue exhibiting varying degrees of inductive changes and
dental hard tissue formation [11, 12].
Radiographically, this tumour varies from a unilocu-
lar or multilocular mixed radio-lucent radiopaque lesion
with irregular size and shape to complete radio opacity as
in the current case consistent with odontoma formation
[13, 14]. Histopathological examination reveals the pres-
ence of island and strands of odontogenic epithelium with
peripheral columnar cells with palisading nuclei resembling
ameloblast and central stellate reticulum-like cells were seen
which resembled ameloblastic follicle. The mesenchymal
component was fibrous and interspersed with large plump
Case Reports in Medicine 3

(a) (b)

Figure 3: Surgical enucleation of the lesion under general anesthesia.

(a) (b)

Figure 4: Histopathological specimen (a) reveals the presence of the lesional tissue in toto along with the attached tooth specimen. (b)
Histopathology shows odontogenic epithelium with peripheral palisading nuclei resembling ameloblast-like cells and loosely arranged central
cells, identical to stellate reticulum, embedded in a myxoid cell-rich stroma resembling the dental papilla. Focal areas of enamel and dentin
are also present.

fibroblast resembling dental papilla. The dentin may vary
structurally from dentinoid to tubular dentin. Our case
histopathological picture was consistent with the above find-
ings [15].
Differential diagnosis of ameloblastic fibroodontoma in-
cludes odontoma, ameloblastoma, ameloblastic fibroma, od-
ontoameloblastoma, calcifying epithelial odontogenic tu-
mour, calcifying epithelial odontogenic cyst, and adenoma-
toid odontogenic tumour. These clinical conditions can be
clearly differentiated from ameloblastic fibroodontoma based
on radiographs due to the presence of dental hard tissue
(enamel and dentin), clinical findings, and prognosis that
also supplemented our case diagnosis [16]. Ameloblastic
fibroodontoma (AFO) includes a conservative surgical enu-
cleation with the removal of associated tooth to prevent any
further recurrences in future as performed in the above case.
However, in few cases, the unerupted tooth left behind has
shown good prognosis.
4. Conclusion
A case of ameloblastic fibroodontoma is rare and unac-
quainted to a clinician due to underreported number of
cases in literature. This article presents an exceptional case
report of ameloblastic fibroodontoma (AFO) with emphasis
on diagnostic criteria thus emphasizing to all the readers
its importance. Treatment of this lesion involves a complete
surgical enucleation with removal of associated tooth. Addi-
tional cases need to be conveyed into certification so as to
have a strong understanding concerning various aspects of
this disease.
Conflicts of Interest
The authors declare that they have no conflicts of interest.
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